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    Neoappendicostomy in the management of pediatric

    fecal incontinence

    Kaveer Chatoorgoon, Alberto Pena, Taiwo Lawal, Miller Hamrick,Emily Louden, Marc A. Levitt

    Colorectal Center for Children, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA

    Received 19 March 2011; accepted 26 March 2011

    Key words:Appendicostomy;

    Neoappendicostomy;

    Fecal incontinence;

    Pediatric;

    Antegrade continence

    enemas

    Abstract

    Purpose:The Malone appendicostomy, for antegrade enemas, has improved the quality of life for many

    children with fecal incontinence. In patients whose appendix has been removed, a neo-appendix can be

    created. We describe our approach and experience with this procedure as an option for surgeons

    managing children with fecal incontinence.

    Methods:The procedure involves creating a transverse flap of cecum that receives its blood supply by a

    transverse mesenteric branch. This flap is then tubularized around a feeding tube. The surrounding colon

    is plicated around the neo-appendix to prevent leakage of stool. The tip of the flap is then anastomosed

    to the deepest portion of the umbilicus. We reviewed our experience with this procedure, including

    results and complications. IRB approval was obtained.

    Results: Eighty patients required a neo-appendicostomy. Sixty-six patients (82%) had an anorectalmalformation, four had spina bifida, and ten had other diagnoses. The reasons for not having an

    appendix available included: incidental appendectomy (34, 42.5%), use of the appendix for a

    Mitrofanoff procedure (20, 25%), and Ladd's procedure (5, 6%). In fifteen patients (19%) we could find

    no appendix and assume that it was removed previously. Following neoappendicostomy, nine patients

    (11%) developed a stricture, and seven patients had leakage (9%). In 2004, we modified the

    appendiceal-umbilical anastomosis and among these patients, only one patient (3%) developed a

    stricture, compared with eight patients (18%) without the modification. All seven patients with leakage

    were within the first forty cases. No patient in the last forty cases had a leakage.

    Conclusions: In patients with the potential for fecal incontinence, the appendix should be preserved. In

    patients without an appendix, the neo-appendicostomy is a valuable tool for fecally incontinent patients.

    We have found that the V-V anastomosis had a reduced rate of stricture, and the rate of leakage seems to

    be related to surgical experience.

    2011 Elsevier Inc. All rights reserved.

    The appendicostomy procedure for antegrade continence

    enemas (ACE), first introduced by Malone [1], has helped

    improve the quality of life for many pediatric patients. In

    particular, the procedure has helped children with anorectal

    malformations, spinal anomalies, and other diagnoses that

    Corresponding author. Department of Surgery, Division of Pediatric

    Surgery, Colorectal Center for Children, Cincinnati Children's Hospital

    Medical Center, University of Cincinnati, Cincinnati, OH 45229, USA.

    Tel.: +1 513 636 3240; fax: +1 513 636 3248.

    E-mail address:[email protected](M.A. Levitt).

    www.elsevier.com/locate/jpedsurg

    0022-3468/$ see front matter 2011 Elsevier Inc. All rights reserved.

    doi:10.1016/j.jpedsurg.2011.03.059

    Journal of Pediatric Surgery (2011) 46, 12431249

    mailto:[email protected]://dx.doi.org/10.1016/j.jpedsurg.2011.03.059http://dx.doi.org/10.1016/j.jpedsurg.2011.03.059mailto:[email protected]
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    suffer from fecal incontinence[2]. In many of these patients

    though, previous procedures have made the appendix

    unavailable or unusable for the appendicostomy procedure.

    Kiely et al described the formation of a neo-appendicostomy

    by using a flap of cecum[3]. We have adopted this approach,

    with some modifications, for children who are fecally

    incontinent and have no appendix available for an ACE.

    The purpose of this study is to describe our approach andreview the outcomes.

    1. Methods

    We reviewed our entire experience with neo-appendicos-

    tomies from October 1994 to September 2009. Data

    extracted included: diagnosis, indication for neoappendi-

    costomy, risk factors for incontinence, ultimate fate of the

    appendix (prompting the need for a neo-appendicostomy),

    complications following the procedure, and long term

    outcomes. Strictures were defined as a narrowing at the

    umbilico-appendicostomy anastomosis that prevented the

    insertion of the feeding tube for the administration of the

    daily antegrade enemas. Leakage was defined as leakage of

    stool through the appendicostomy between enema adminis-

    trations, which was significant enough to warrant a redo

    operation. IRB approval was obtained prior to commencing

    the review.

    1.1. Surgical technique

    Once a child has been diagnosed as fecally incontinent in

    our center, he or she undergoes a bowel management programwith daily enemas to keep him/her mechanically clean of

    stool[4-6]. After we have demonstrated that the patient can

    remain clean, we discuss the possibility of antegrade colonic

    enemas, which provides the patient with a more comfortable

    and independent route for enema administration. If the patient

    has had a previous appendectomy, then our approach is to use

    a neo-appendicostomy, using a flap of colon.

    The day prior to surgery, the patient undergoes a bowel

    preparation. The patient is placed under anesthesia and is

    positioned supine. The procedure begins, as it would for a

    Malone appendicostomy, by creating a triangular flap in the

    umbilical skin for the eventual appendiceal-umbilical anas-

    tomosis (Fig. 1). Once the flap has been created, an

    infraumbilical midline laparotomy is performed from the

    umbilicus down. The right colon is identified. If no appendix

    is present, the right colon is mobilized off of its retroperito-

    neal attachments, in preparation for the neo-appendicostomy.

    Fig. 1 Triangular flap of skin is created at the umbilicus in preparation for the anastomosis.

    1244 K. Chatoorgoon et al.

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    The colon is inspected for an appropriate location for the

    flap, usually in the ascending colon, away from the ileocecal

    valve, with two feeding vessels from the mesentery (Fig. 2).

    The flap must be large enough that it will easily tubularize

    around an 8F feeding tube. Taking care not to injure the blood

    supply, the flap is created and reflected away (Fig. 3). The

    flap is then closed over an 8F feeding tube, simultaneously

    closing the colon in the transverse direction, in two layers

    (Fig. 4). The neo-appendix is then laid down and the colonic

    wall is plicated (wrapped) around it. Special care is taken toavoid leaving the suture line of the neo-appendix in contact

    with the colonic suture line, to avoid the risk of a fistula. This

    plication must be tight enough to prevent leakage, but not too

    tight that the feeding tube cannot pass through. We perform

    the plication with the feeding tube in place (Fig. 5), and pass

    the tube to check patency after each plication stitch.

    The umbilical-appendiceal anastomosis is then per-

    formed. During our initial experience with appendicostomies

    and neo-appendicostomies, the tip of the appendix was

    sutured to the base of the umbilicus, forming a circular

    anastomosis. This early experience had a high rate of

    stricture formation, leading us to a change the type of

    anastomosis to one that involves a triangular flap of skin (V

    to V anastomosis) (Fig. 6). The tip of the neo-appendix is left

    open, forming a V. The appendix is then positioned such

    that the triangular skin flap (created at the beginning of thelaparotomy) will be anastomosed to the base of the V.

    Using fine 6-0 vicryl sutures, each side of the triangular skin

    flap is anastomosed to either side of the appendiceal incision.

    At the end, the triangular flap forms one part of the orifice, as

    the floor of the tunnel, through which the catheter will be

    passed (Figs. 6, 7).

    Fig. 2 Flap of cecum created on a pedicled blood supply from the mesentery. The flap is then rotated out and closed around a feeding tube.Reprinted with permission from Elsevier[2].

    Fig. 3 Flap of cecum reflected out prior to tubularization.

    1245Neoappendicostomy in managing fecal incontinence

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    Fig. 4 Closure of the flap over an 8F feeding tube. The cecal defect is closed at the same time.

    Fig.5 The neo-appendix is then wrapped by a segment of colon, creating a valve mechanism to minimize leakage. Illustration reprinted with

    permission from Elsevier[2].

    1246 K. Chatoorgoon et al.

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    The patient usually stays in the hospital for 2 to 4 days.

    Once on a regular diet, we begin half volume enemas twice

    per day, through the neo-appendi costomy. They are

    discharged with the 8F feeding tube secured in place for a

    month. One month post-operatively, the catheter is removed

    and the family is taught how to introduce the tube through

    the healed neo-appendicostomy. Then the patient is placed

    on their routine enema, once daily.

    2. Results

    Eighty patients underwent a neo-appendicostomy at an

    average age of 11.7 years (47 were male, 33 female). Sixty-

    six patients (82%) had an anorectal malformation, 4 patients

    had spina bifida (5%), 3 had Hirschsprung disease (4%), and

    2 had idiopathic constipation (2.5%). Five additional patients

    (7%) had the following diagnoses: presacral cystic teratoma,

    myelomeningocele, caudal regression, Currarino Triad, and

    developmental delay secondary to a chromosomal anomaly.Seventy-six patients (95%) were truly fecally-incontinent.

    Four patients had severe constipation requiring daily enemas

    because they were unmanageable on laxatives (5%).

    In all cases, the appendix was either previously resected,

    or not usable for the appendicostomy. Thirty-four patients

    (42.5%) had undergone an incidental appendectomy

    during a previous procedure. In 20 patients (25%), the

    appendix was used in a Mitrofanoff procedure. In 15 patients

    (19%) we could find no appendix, and assumed that it was

    removed incidentally during one of the previous operations,

    even though it was not noted in the operative report, nor told

    to the family. Five patients (6%) had a failed Malone

    procedure performed previously at an outside institution, and

    during the redo operation, the appendix was found to be

    unusable. Five patients (6%) underwent a Ladd's procedure

    in which the appendix was removed as the routine part of the

    procedure, and one patient had an appendix with an

    obliterated lumen. In 5 patients, we converted a button

    cecostomy created elsewhere, into a neo-appendicostomy.

    The majority of patients had their neo-appendicostomy

    placed in the cecum or ascending colon (66 patients). Three

    patients had it placed in the sigmoid, 2 in the transverse

    colon, and 1 in the descending colon.

    Fig. 6 The V-V Umbilical-Appendiceal Anastomosis. This anastomosis incorporates umbilical skin to minimize stricture formation.

    Fig. 7 The final appearance shows the stoma is hidden within the

    umbilicus. The 8F feeding tube is sutured in place, and left in the

    lumen of the neo-appendicostomy. Reprinted with permission from

    Levitt MA, Pea A. Laparoscopy in the Management of Fecal

    Incontinence and Constipation, In: Atlas of Pediatric Laparoscopy

    and Thoracoscopy. Holcomb, Georgeson, Rothenberg, eds. Saun-

    ders Elsevier, 2008, pp. 81-90.

    1247Neoappendicostomy in managing fecal incontinence

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    Following neoappendicostomy, 9 patients (11%) deve-

    loped a stricture and 7 patients had leakage (9%). No patient

    suffered from an anastomotic dehiscence. All of these

    patients required a redo operation to correct the problem,

    which resulted in an overnight stay in the hospital. In 2004,

    we modified the appendiceal-umbilical anastomosis to utilize

    a triangular flap of umbilical skin, in an attempt to reduce

    the rate of stricture formation. In the 36 patients who hadthis V-V anastomosis, only 1 patient (3%) developed a

    stricture, compared with 8 patients (18%) without the V-V

    anastomosis (P= .04). All 7 cases with leakage occurred

    within the first 40 cases; no patient in the last 40 cases had

    a leakage. Two patients had ischemia of their neoappendi-

    costomy requiring urgent reoperation. In one of these

    patients, the neoappendicostomy was redone, and in the

    other, it was converted to a button cecostomy. Neither of

    these patients required admission to the intensive care unit.

    Two patients had a post-operative bowel obstruction that

    resolved spontaneously. Three patients were lost to follow-

    up. Currently, 70 of the 77 (91%) patients available for

    follow-up clean with their enema regimen; while sevenpatients require their regimens to be adjusted to optimize

    their social continence.

    3. Discussion

    Bowel management with enemas provides fecally incon-

    tinent patients with a method of staying clean of stool in their

    underwear for 24 hours [4-6]. Rectal enemas are easily

    administered to small children. However, as they grow up, it

    becomes more difficult, as they are difficult to administer bythe patient alone, and usually requires the aid of a parent or

    caregiver. The Malone appendicostomy provides indepen-

    dence, as older children can then manage their enemas

    without assistance. The neoappendicostomy attempts to

    provide this same level of independence in patients who have

    had an appendectomy, or whose appendix was used for

    another purpose. A stoma hidden within the umbilicus

    presents a nice cosmetic option.

    Due to our specialized practice, the vast majority of the

    patients had anorectal malformations. Many of these

    patients have had multiple prior operations, including

    urologic reconstructions, and so they may have lost their

    appendix incidentally, or it was used as part of a urinary

    continent diversion (Mitrofanoff procedure). In such

    patients, the neo-appendicostomy procedure has become

    our mainstay procedure.

    The two main complications we encountered were

    stricture formation and leakage. We have found a statistically

    significant decrease in the incidence of stricture formation

    with the implementation of the V to V triangular flap into the

    anastomosis. We believe that this flap of skin acts like a

    wedge, keeping the edges of appendiceal mucosa from

    healing together and narrowing. In terms of leakage, the

    degree of plication appears to be the most important factor.

    There is certainly a balance between making the plication too

    tight, and risking compromise to the blood supply, and

    making it too loose and risking leakage. We believe that the

    two cases of necrosis were due to compression of the blood

    supply by a tight plication. The rate of leakage following

    neo-appendicostomy has decreased as our experience has

    grown; in the last half of our series, no leakages haveoccurred. Perhaps this learning curve explains the reduction

    in this complication.

    Our approach is different than Kiely's original descrip-

    tion in several ways [3]. We form our flap on the

    mesenteric, rather than antimesenteric, side of the bowel.

    We prefer this position as the mesenteric vessels feed the

    flap directly, rather than through intramural collateral flow.

    In order to limit leakage, we plicate the cecum around the

    base of the neoappendicostomy. Additionally, we attach the

    neoappendicostomy to the base of the umbilicus, to hide the

    stoma for better cosmesis. Finally, we perform a V to V

    anastomosis to reduce stricture. We have found that these

    modifications to Kiely's technique have helped to reducethe number of complications.

    The button cecostomy is a common alternative for

    patients with no appendix available. These buttons can be

    placed open[7], laparoscopically[8], percutaneously[9]and

    endoscopically[10]. We have noted an increased number of

    patients attending our bowel management clinic, who have

    had this procedure done elsewhere. The reported advantages

    of the button cecostomy include a shorter operative time, and

    the ability to perform the procedure laparoscopically[11,12].

    Noted complications include dislodgement, granulation

    tissue formation, leakage, pain, and ulcers (from pressure

    necrosis of the tube) and occur in nearly 50% of the patients[11-13]. Still, many of these complications can be managed

    in the clinic with simple maneuvers, including replacement

    and resizing. Many of these studies also show a subjective

    improvement in quality of life, but it is difficult to know if

    the improvement is due to the success of the antegrade

    colonic enemas, or to the button cecostomy itself. Our

    experience is that these tubes create foreign body reactions,

    similar to gastrostomy tubes, with leakage of stool,

    granulation tissue, and pain. In fact, Cascio et al warns the

    parents preoperatively, that leaking around the button is

    expected and that granulation tissue can develop in one third

    of cases[13]. We have removed several cecostomy buttons

    (3 in this series) at the request of the patient and family,

    because of these symptoms, and converted them to neo-

    appendicostomies. Anecdotally, these patients and families

    have preferred the cosmetic result of a neoappendicostomy to

    the button.

    Interestingly, none of the patients in this series had their

    appendix removed for appendicitis. Over half the patients

    (n = 49) had their appendix removed incidentally. As a rule,

    we avoid incidental appendectomies in patients with risk

    factors for fecal incontinence (for example, anorectal

    malformation patients with prostatic or bladder neck fistulae,

    1248 K. Chatoorgoon et al.

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    poorly formed sacrums and tethered cords), even when

    performing a Ladd's procedure. We advocate against

    incidental appendectomies in these high-risk patients in

    case they turn out to require bowel management.

    In 19 patients, the appendix was used in a Mitrofanoff

    procedure. Often, the appendix can be split and used for both

    the Mitrofanoff and Malone procedures [14]. With this in

    mind, we coordinate with the urologists regarding patientswith urologic issues and fecal incontinence, to perform the

    Mitrofanoff procedure in conjunction with the Malone

    procedure, during the same operation.

    4. Conclusion

    The neo-appendicostomy is a valuable tool in the

    management of fecally incontinent patients who have had

    their appendix resected or reconstructed. We have found that

    the V to V triangular anastomosis reduces the risk of stricture

    and that the risk of leakage decreases with experience. If atall possible, patients with the possibility of fecal inconti-

    nence should have their appendix preserved, to allow for the

    potential for an appendicostomy procedure in the future.

    References

    [1] Malone PS, Ransley PG, Kiely EM. Preliminary report: the antegrade

    constinence enema. Lancet 1990;336:1217-8.

    [2] Levitt MA, Soffer SZ, Pena A. Continent appendicostomy in the bowel

    management of fecally incontinent children. J Pediatr Surg 1997;32:

    1630-3.

    [3] Kiely EM, Ade-Ajayi N, Wheeler RA. Caecal flap conduit for

    antegrade continence enemas. Br J Surg 1994;81:1215.

    [4] Pena A, Guardino K, Levitt MA. Bowel management for fecal

    incontinence in patients with anorectal malformations. J Pediatr Surg

    1998;33:133-7.

    [5] Bischoff A, Levitt MA, Pena A. Bowel management for the treatment

    of pediatric fecal incontinence. Pediatr Surg Int 2009;25:1027-42.[6] Bischoff A, Tovilla M. A practical approach to the management of

    pediatric fecal incontinence. Semin Pediatr Surg 2010;19:154-9.

    [7] Duel BP, Gonzalez R. The button cecostomy for management of fecal

    incontinence. Pediatr Surg Int 1999;15:559-61.

    [8] Yagmurlu A, Harmon CM, Georgeson KE. Laparoscopic cecostomy

    button placement for the management of fecal incontinence in children

    with Hirschsprung's disease and anorectal anomalies. Surg Endosc

    2006;20:624-7.

    [9] Sierre S, Lipsich J, Questa H, et al. Percutaneous cecostomy for

    management of fecal incontinence in pediatric patients. J Vasc Interv

    Radiol 2007;18:982-5.

    [10] Miyani D,BaroowE,HodsonP, etal. Endoscopicallyplacedcaecostomy

    buttons:a trialACEprocedure.Colorectal Dis 2007;9:373-6.

    [11] Becmeur F, Demarche M, Lacreuse I, et al. Cecostomy button for

    antegradeenemas: surveyof 29 patients.J PediatrSurg 2008;43:1853-7.[12] Wong AL, Kravarusic D, Wong SL. Impact of cecostomy and

    antegrade colonic enemas on management of fecal incontinence and

    constipation: ten years of experience in pediatric population. J Pediatr

    Surg 2008;43:1445-51.

    [13] Cascio S, Flett ME, De la Hunt M, et al. MACE or caecostomy button

    for idiopathic constipation in children: a comparison of complications

    and outcomes. Pediatr Surg Int 2004;20:484-7.

    [14] Kajbafzadeh AM, Chubak N. Simultaneous Malone antegrade

    continent enema and Mitrofanoff principle using the divided appendix:

    report of a new technique for prevention of stoma complications.

    J Urol 2001;165:2404-9.

    1249Neoappendicostomy in managing fecal incontinence